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Giant cystadenoma of the floor of the mouth: A case report
Victor Labres da Silva Castro1, Tiago Fernando Aires Corrêa2, Valeriana de Castro Guimarães3, Gustavo Vasconcelos Nery2,João Batista Ferreira4.
1) Doctor, Otolaryngology resident.2) Otolaryngologist.3) Health Science PhD student, Speech Therapist, Clinical Hospital - UFG.4) Postdoctoral Fellow in Otolaryngology. Associate Professor, Department of Surgery, Faculty of Medicine-UFG. Head of Clinical Otorhinolaryngology-UFG.
Institution: Hospital of the Federal University of Goiás.Goiânia / GO - Brazil.
Mailing address: Otorrinolaringologia - Hospital das Clínicas da Universidade Federal do Goiás - Primeira Avenida, s/n - Setor Leste Universitário – Zip code:74605-020 - Goiânia / GO - Brazil - Telephone: (+55 62) 3269-8387 - E-mail: [email protected] received August 25, 2010. Article accepted February 5, 2011.
SUMMARY
Introduction: Cystadenoma is an uncommon epithelial neoplasia that arises from the salivary glands. The malignancy can affect
structures such as the larynx, nasopharynx, buccal mucosa, and palate.
Objective: To describe a case of a giant cystadenoma of the floor of the mouth treated at a public hospital in midwestern Brazil.
Case report: The patient was a 46-year-old woman with complaints of difficulties in articulating words and swallowing solid
food and vocal fatigue. The progression of the disease since the initial consultation, the results of clinical examinations, and
the outcome of surgery are described.
Finals Comments: Cystadenoma must be considered in the differential diagnosis of cystic injuries in the floor of the mouth
if the patient’s symptoms are suggestive of this malignancy.
Keywords: salivary glands, minor; mouth; mouth mucosa; exocrine glands.
INTRODUCTION
The salivary glands are formed by a set of exocrine
glands that together excrete their secretions into the oral
cavity (1). Tumors in this region are rare and of varied
etiology, and they account for approximately 3% of all
lesions in this region (2,3,4).
Benign tumors represent 75% of the lesions affecting
salivary glands. The majority of these disorders (70%)
appear in the parotid gland, with 22% appearing in the
submandibular gland and 8% appearing in the minor
salivary glands (4). There is no association between smoking
and benign tumors of the salivary glands (4).
Cystadenoma is an uncommon epithelial neoplasm
that arises from the salivary glands (5). The disease appears
beginning in the 5th decade of life, with both men and
women affected alike (2,3,4). The malignancy may affect
structures such as the larynx, nasopharynx, oral mucosa,
and palate; however, their manifestations are asymptomatic
and painless (4).
This paper presents a relevant and unusual case in
otorhinolaryngology distinguished by the rarity of its
occurrence and the large size of its lesion.
In this report, we describe the case of a patient with
a giant cystadenoma of the floor of the mouth who visited
the Hospital of the Federal University of Goiás in Goiânia.
CASE REPORT
The patient was a 46-years-old woman born in
Damianápolis (GO) who visited the otorhinolaryngology
emergency department of the Hospital of the Federal
University of Goiás HC/UFG in June 2006 with the following
complaints: difficulty in articulating words, difficulty in
swallowing solid foods, and vocal fatigue. No other
otorhinolaryngology symptoms were identified.
The patient reported a worsening of the problem in
recent months due to the increased volume of the floor of
the mouth, which was affecting the swallowing of solid
food. She also reported dissatisfaction with her current
facial features and iron deficiency anemia due to the
undertreatment of hypermenorrhea.
At the initial consultation, the patient had a good
general condition with normal vital signs. On inspection
and cervicofacial palpation, a volume increase in the
submental and sublingual region of elastic-firm consistency
that was painless to finger compression was identified. No
Case Report Int. Arch. Otorhinolaryngol. 2013;17(1):101-104.
DOI: 10.7162/S1809-97772013000100018
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alterations in the neck were observed, and the absence of
cervical lymphadenomegaly was confirmed. The presence
of facial deformity characterized by protrusion of the
submental and sublingual region was also identified.
In the previous clinical evaluation, both
rhinolaryngoscopy and otoscopy revealed no changes. Via
oropharyngoscopy, we observed a large mass of the cystic
aspect occupying the entire floor of the mouth (Figure 1),
with compression of the tongue toward the craniodorsal
direction and without visualization of Wharton’s duct papillae.
Nasopharyngolaryngoscopy revealed posterior
displacement of the tongue base. No alterations were
observed in the right nasal cavity, pharynx, or larynx.
Computed tomography (CT) scanning of the
hemiface revealed a benign cystic lesion in the mouth floor
with a hypodense, rounded appearance causing the pos-
terior-superior displacement of the tongue. Magnetic
resonance imaging (MRI) identified a bulky cystic mass in
the supramandibular region (Figure 2).
In 2008, after prolonged hematologic treatment of
anemia, the patient underwent surgery. With the diagnosis
of a cystic lesion in the mouth floor, we opted for an
intraoral surgical approach. After performing an accurate
anterior incision in the mucosa of the mouth floor with
careful dissection of the cystic capsule and complete
excision of the lesion (Figure 3), approximation of the
partially separated muscles and suture of the mucosa were
performed with the placement of a drain for 24 h and a
nasogastric tube for 3 days. In the immediate postoperative
period, the patient displayed mild sublingual edema that
was treated with prednisone 20 mg/day for 5 days.
Histopathological examination identified a cystic
lesion lined by ciliated, cylindrical, flattened epithelium
with atrophic areas separated by seromucous glands and
papillary intracystic extensions. The benign cystic lesion
originating in the minor salivary glands was comparable to
a cystadenoma.
DISCUSSION
The interest in the description of this case is
evidenced by the rarity of its occurrence as well as the
conduct and outcome of the same. Several studies of
cystadenoma have been performed in various areas of
health. However, no studies similar to this study have been
published.
The floor of the mouth can be compromised by a
variety of injuries; many are major challenges to
Figure 1. Patient with bulky lesion bulging floor of the mouth
HC / UFG 2006.
Figure 2. Nuclear Magnetic Ressonâncio. 2006.
Figure 3. Surgical removal of the cyst. Capsule integrates. HC
/ UFG 2008.
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Giant cystadenoma of the floor of the mouth: A case report. Castro et al.
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professionals, leading to misdiagnosis and incorrect
treatment (6).
Usually, cystic lesions located in this region are
benign, and they arise from the salivary glands and are of
varied origin, ranging from embryologic to neoplastic.
They are characterized by insidious and progressive growth,
and symptoms occur late in the pathogenesis when lesions
reach a high volume (3,4,7). Similar to findings described
in the literature, the patient sought medical attention years
after the lesion developed, probably owing to the absence
of symptoms.
Lesions in this region should be submitted to
differential diagnosis to exclude the presence of other
diseases such as ranula, neoplasms of the major and minor
salivary glands, cystic hygroma, dermoid cysts, lipoma, and
cellulite (7).
Cystadenoma is a rare benign epithelial neoplasm
that commonly occurs in the parotid and minor salivary
glands and affects the oral and labial mucosa (5). In our
case, the tumor appeared on the floor of the mouth. In
this region, the slow growth of tumors may delay
medical care, which can lead to significant injuries
resulting in the appearance of anatomical and functional
changes such as alterations in speech, swallowing
disorders, dyspnea, and cosmetic deformities, similar to
the presented case.
Although the average age of patients diagnosed
with cystadenoma is approximately 50 years, the literature
also includes cases of patients with congenital cystadenomas
(5,8). In this case, the patient reported that the cystadenoma
had been present since childhood, which reinforces the
hypothesis of a congenital cyst.
The diagnosis of the disease is based on the clinical
evaluation of the lesion; however, imaging tests are useful
for determining differences between cysts and solid tumors
and demonstrating the location and extent of lesions,
assisting in defining the clinical and surgical approach.
However, these tests do not differentiate between benign
and malignant lesions (9,10).
CT and MRI have similar sensitivity and specificity
for assessing the location and infiltration of the tumor mass,
and they can be performed to assess the involvement of
deep spaces or contralateral glands (10). Prior knowledge
of the boundaries and dimensions of the cyst in relation to
the anatomy of the mouth floor are important aspects to be
considered when planning surgery (7). In the present case,
MRI identified a bulky, rounded, hypodense mass
compatible with cystic lesions that caused posterior-supe-
rior displacement of the tongue. Thus, the proposed
surgery considered the results of imaging tests performed
preoperatively.
Benign tumors of the salivary glands are treated by
surgical excision of the lesion, the purpose of which is to
correct cosmetic deformities and prevent complications
such as nerve compression or malignant progression (4,5,8).
The intraoral surgical approach is recommended for all
cases of tumors in this region (7). In our patient, intraoral
surgery was performed with careful and complete dissection
of the lesion without damage to adjacent structures.
FINAL COMMENTS
Cystadenoma should be considered in the
distinguishing diagnosis of cystic lesions in the floor of the
mouth if the clinical findings are suggestive of this disease.
The participation and cooperation of the patient, including
attending all consultations and adhering to treatment,
contribute to successful treatment.
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Disponível em http://www.emedicine.com/ent/
topic679.htm.
Int. Arch. Otorhinolaryngol., São Paulo - Brazil, v.17, n.1, p. 101-104, Jan/Feb/March - 2013.
Giant cystadenoma of the floor of the mouth: A case report. Castro et al.